The impact of craniofacial and dental osteogenesis imperfecta manifestations on oral health-related quality of life of children and adolescents

Abstract

Objective: Craniofacial and oral manifestations of Osteogenesis Imperfecta (OI) can affect the functioning of the stomatognathic system and impact the patient's quality of life. The objective of the study was to evaluate the relationship between craniofacial and oral manifestations and the Oral Healthrelated Quality of Life (OHRQoL) of OI children and adolescents. Material and Methods: A total of 30 OI patients aged 8 to 14 years old followed up at the Oral Care Center for Inherited Diseases were enrolled the research. OHRQoL was assessed using the of the Child Perceptions Questionnaire (CPQ) for 8 to 10-year-olds (CPQ) and for 11 to 14-year-olds ). The relationship between the OHRQoL index and its assessment domains, OI types, and the presence of dentinogenesis imperfecta (DI), class III malocclusion and dental agenesis were evaluated.Results: The median CPQ score of patients was 5 and there w no signi cant difference on OHRQoL between children and adolescents, nor associated with the disease severity or the presence of DI. The oral manifestations evaluated did not directly impact the patients' OHRQoL.Conclusions: The study demonstrated that perception of OHRQoL is similar for both adolescents and children. The oral symptom was the most relevant domain for the index among patients aged 8 to 14 years while the well-being was the most impacted.Clinical Relevance: The detailed assessment of OHRQoL in OI patients helps in the development of more quali ed treatment strategies focused on the factors with the greatest impact on the patient's life.